Klossowski N, Braun S A, von Gruben V, Losem C, Plewe D, Homey B, Meller S
Hautklinik des Universitätsklinikums Düsseldorf, Moorenstr. 5, 40225, Düsseldorf, Deutschland.
Praxis Hämatologie und Onkologie Neuss, Neuss, Deutschland.
Hautarzt. 2015 Oct;66(10):723-5. doi: 10.1007/s00105-015-3679-9.
Acquired angioedema due to C1 inhibitor deficiency (C1-INH-AAE) is characterized by recurrent edema of the subcutaneous and/or submucosal tissue without wheals and negative family history of angioedema. Here, we present the case of a patient with a chronic lymphatic B cell leukemia who suffered from both C1-INH-AAE and chronic spontaneous urticaria. Oral corticosteroids, antihistamines, and the anti-IgE antibody omalizumab were applied to treat the chronic urticaria in combination with the plasma-derived C1 esterase inhibitor concentrate Berinert® and the bradykinin B2 receptor antagonist icatibant, but the symptoms did not improved significantly. Thus, polychemotherapy targeting the slow-growing lymphoproliferative disease including rituximab was initiated, which resulted in remission of both the urticaria and the angioedema.
由于C1抑制剂缺乏所致的获得性血管性水肿(C1-INH-AAE)的特征是皮下和/或粘膜下组织反复出现水肿,无风团,且血管性水肿家族史阴性。在此,我们报告一例慢性B淋巴细胞白血病患者,其同时患有C1-INH-AAE和慢性自发性荨麻疹。口服糖皮质激素、抗组胺药以及抗IgE抗体奥马珠单抗用于治疗慢性荨麻疹,同时联合使用血浆源性C1酯酶抑制剂浓缩物贝林妥欧®和缓激肽B2受体拮抗剂依卡替班,但症状改善不明显。因此,启动了针对生长缓慢的淋巴增殖性疾病的多药化疗,包括利妥昔单抗,这导致荨麻疹和血管性水肿均得到缓解。